Association of the family environment with behavioural and cognitive outcomes in children with chromosome 22q11.2 deletion syndrome

Abstract

Background Children with 22q11.2 deletion syndrome (22q11DS) are at risk for social-behavioural and neurocognitive sequelae throughout development. The current study examined the impact of family environmental characteristics on social-behavioural and cognitive outcomes in this pediatric population. Method Guardians of children with 22q11DS were recruited through two medical genetics clinics. Con senting guardians were asked to complete several questionnaires regarding their child's social, emotional and behavioural functioning, as well as family social environment and parenting styles. Children with 22q11DS were asked to undergo a cognitive assessment, including IQ and achievement testing, and measures of attention, executive function and memory. Results Modest associations were found between aspects of the family social environment and parenting styles with social-behavioural and cognitive/academic outcomes. Regression models indicated that physical punishment, socioeconomic status, parental control and family organisation significantly predicted social-behavioural and cognitive outcomes in children with 22q11DS. Conclusion Characteristics of the family social environment and parenting approaches appear to be associated with functional outcomes of children with 22q11DS. Understanding the impact of environmental variables on developmental outcomes can be useful in determining more effective targets for intervention. This will be important in order to improve the quality of life of individuals affected by 22q11DS.

Understanding the pediatric psychiatric phenotype of 22q11.2 deletion syndrome

“…Several studies point towards the impact of environmental factors on the observed behavioral phenotype in individuals with 22q11DS. For example, studies have shown that lower parental socio-economic-status (SES) and intrusive parenting style [Barber 2002] were associated with worse social functioning and other clinically significant problems in children with 22q11DS [Allen and others 2014; Shashi and others 2010]. Additional evidence is provided by a study examining the effect of Sept5 deficiency (one of the genes in the 22q11.2 region) on social functioning in a mice model of 22q11DS[Harper and others 2012].…”

Childhood Predictors of Young Adult Social Functioning in 22q11.2 Deletion Syndrome

“…Parents of children with 22q11.2DS also report experiencing marital conflict and having lower than average expectancies for their children for functional independence and academic achievement, thereby requiring more close supervision (Allen et al, 2014; Prinzie et al, 2004). Moreover, family environment can influence social functioning (e.g., modeling how to resolve conflicts) in typically developing adolescents (Youngblade et al, 2007).…”

Association Between Parental Anxiety and Depression Level and Psychopathological Symptoms in Offspring With 22q11.2 Deletion Syndrome

“…Nevertheless, the study of other important environmental factors, such as specific aspects of the familial environment, has received limited attention. The association between parenting style and behavioral outcome in the offspring diagnosed with 22q11DS has been investigated in two studies (10,11), both of them showing significant associations. Weisman et al (12) also investigated dyadic reciprocity during a mother-child interaction and showed significant associations with the level of behavior problems assessed with the Child Behavior Checklist [CBCL (13)].…”

“…Nevertheless, the study of other important environmental factors, such as specific aspects of the familial environment, has received limited attention. The association between parenting style and behavioral outcome in the offspring diagnosed with 22q11DS has been investigated in two studies ( 10 , 11 ), both of them showing significant associations. Weisman et al.…”